{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T05:31:07Z","timestamp":1788413467878,"version":"build-2803163510"},"reference-count":79,"publisher":"The Endocrine Society","issue":"5","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2018,5,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Context<\/jats:title>\n                    <jats:p>Management of congenital adrenal hyperplasia (CAH) involves suppression of the hypothalamic\u2013pituitary\u2013adrenal axis using supraphysiological doses of exogenous glucocorticoids. This can pose a challenge, with Cushing syndrome a frequent complication of adequate suppression. Bilateral adrenalectomy, with subsequent replacement of glucocorticoids and mineralocorticoids at physiological doses, has been proposed as an alternative therapeutic strategy.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To review the outcomes after bilateral adrenalectomy for CAH.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Data Sources<\/jats:title>\n                    <jats:p>A systematic search of PubMed\/MEDLINE and Web of Science, identifying relevant reports published up to 10 January 2018.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Study Selection<\/jats:title>\n                    <jats:p>Case reports or case series were included if they reported individual patient data from patients with CAH who had undergone bilateral adrenalectomy.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Data Extraction<\/jats:title>\n                    <jats:p>Information regarding the following was extracted: first author, country, sex, age at adrenalectomy, year of adrenalectomy, diagnosis, molecular abnormality, pre- and postoperative biochemistry, pre- and postoperative medications, pre- and postoperative body mass index, indication for adrenalectomy, surgical technique, gross and microscopic adrenal characteristics, follow-up duration, and short- and long-term postoperative outcomes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Data Synthesis<\/jats:title>\n                    <jats:p>We identified 48 cases of bilateral adrenalectomy for CAH, with patients aged from 4 months to 56 years at surgery. The most common indication for surgery was the inability to control hyperandrogenism\/virilization and\/or Cushing syndrome (n = 30; 62%). Most patients (n = 34; 71%) reported symptomatic improvement postoperatively, with some cases of short-term (n = 5; 10%) and long-term (n = 13; 27%) adverse outcomes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Bilateral adrenalectomy for CAH appears to be a reasonable therapeutic option for carefully selected patients who have had unsatisfactory outcomes with conventional medical management.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1210\/jc.2018-00217","type":"journal-article","created":{"date-parts":[[2018,3,15]],"date-time":"2018-03-15T14:03:04Z","timestamp":1521122584000},"page":"1767-1778","source":"Crossref","is-referenced-by-count":54,"title":["Bilateral Adrenalectomy in Congenital Adrenal Hyperplasia: A Systematic Review and Meta-Analysis"],"prefix":"10.1210","volume":"103","author":[{"given":"Diana","family":"MacKay","sequence":"first","affiliation":[{"name":"Department of Endocrinology, Royal Darwin Hospital, Darwin, Northern Territory, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Nordenstr\u00f6m","sequence":"additional","affiliation":[{"name":"Department of Women\u2019s and Children\u2019s Health, Karolinska Institutet, Stockholm, Sweden"},{"name":"Department of Pediatric Endocrinology, Astrid Lindgren Children\u2019s Hospital, Karolinska University Hospital, Stockholm, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Henrik","family":"Falhammar","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Royal Darwin Hospital, Darwin, Northern Territory, Australia"},{"name":"Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, Stockholm, Sweden"},{"name":"Department of Molecular 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